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Requisitos metódicos del diseño institucional

EL MÉTODO COMPARATIVO

V. Contexto y diseño institucional

2. Requisitos metódicos del diseño institucional

Summary of Findings

This project examined the potential role of the extracellular matrix protein,

Slit2 and its receptor Roundabout (Robo) in reducing complications following

neurosurgical injury. We specifically examined the contribution of Slit2 in

reducing neuroinflammation and blood brain barrier (BBB) disruption after

surgical brain injury (SBI) using a rat model. The major observations and

conclusions obtained from the project have been summarized in this section.

Endogenous Slit2 has a Protective Function After SBI

Our findings from Chapter 3 show that endogenous Slit2 has a protective

function after SBI. We observed that endogenous Slit2 was upregulated after SBI

starting at 24 h upto 7 days after the injury, and knockdown of endogenous Slit2

using siRNA worsened brain edema and neurological function after SBI. In

addition, we observed that Slit2 was expressed by neurons and astrocytes after

the injury, which we speculate is the source for increase in endogenous levels of

Slit2 as a protective response to injury. These findings complement previous

study in which Slit2 was shown to be increased after traumatic brain injury

(Hagino et al., 2003). Although the function of endogenous Slit2 in adults after

brain injury has not been explored, our findings that endogenous Slit2

knockdown worsened outcomes after SBI suggests a protective role of Slit2

Recombinant Slit2 Reduces SBI Induced Complications

Our findings from Chapter 3 show that recombinant Slit2 pretreatment

reduced SBI induced brain edema and neurological deficits in SBI rats. The

improved outcomes with recombinant Slit2 pretreatment was associated with

reduced neuroinflammation and peripheral leukocyte migration to the perisurgical

site after SBI. Post-operative brain edema is one of the major complications

encountered in neurosurgical patients (Wong et al., 2012). Neuroinflammation

and disruption of the BBB can worsen brain edema and post-operative

neurological function (Bruder and Ravussin, 1999, Hyong et al., 2008, Huang et

al., 2015), which were both attenuated with recombinant Slit2. Additionally, our

results from Chapter 4 show that recombinant Slit2 reduced BBB permeability

and stabilized the BBB tight junction at the perisurgical site after SBI. These

findings support the therapeutic potential of recombinant Slit2 to reduce

neuroinflammation and BBB disruption following brain injury.

Robo Receptor Mediated Signaling Pathways Contribute to the Protective Effects of Slit2

The Robo family of receptors is known to be activated by the Slit2 ligand

(Guan and Rao, 2003, Ballard and Hinck, 2012). We evaluated the role of Robo1

and Robo4 receptors in Slit2 mediated protection after SBI. Our findings in

Chapter 3 show that Robo1 was expressed by peripheral immune cells that

infiltrated at the perisurgical site after SBI. Our results suggest that inactivation of

Cdc42 via Robo1-srGPA1 pathway contributed to the inhibition of peripheral

reversed the anti-migratory effect of recombinant Slit2 on the peripheral immune

cells. Likewise, our findings in Chapter 4 show that Robo4 was expressed by the

endothelial cells. The BBB protective function of recombinant Slit2 was

associated with Robo4-paxillin mediated Rac1 activation, and knockdown of

Robo4 or paxillin reversed Slit2 mediated Rac1 activation. These findings

suggest that Robo1 and Robo4 receptors are primarily involved in mediating the

protective effects of Slit2 by modulating the downstream signaling pathways.

Scientific Contribution

This project was focused on studying SBI, which is an important clinical

problem that has largely been overlooked due to a lack of understanding of the

occurrence of the problem. Even though adverse events such as brain edema

and post-operative neurological deficits often occur following neurosurgical

procedure (Bruder and Ravussin, 1999, Rolston et al., 2014), the

pathophysiology of SBI is often not studied. This project explored two major

pathophysiological events that occur after SBI. First, the role of peripheral

immune cell migration in mediating neuroinflammation after SBI was examined

particularly with a focus on Robo1 mediated immune cell migration to the injury

site. Second, post-operative brain edema resulting from BBB junction disruption

was examined with a focus on restoring BBB stability after SBI.

This project has identified novel molecular mechanisms involved in Slit2

mediated neuroprotection. Slit2 has primarily been studied in the developing

nervous system and few studies have explored the role of Slit2 in adult brain

shown that Slit2 reduced injury outside the CNS in animal models of renal and

lung inflammation (Kanellis et al., 2004, Ye et al., 2010), the function of Slit2 after

brain injury has been relatively unexplored. Slit2 was shown to be increased after

experimental traumatic brain injury (Hagino et al., 2003) and exogenous Slit2

was beneficial in a cerebral ischemia rodent model (Altay et al., 2007). However,

functional significance of Slit2 after brain injury and the protective mechanism of

Slit2 were not determined. Our findings in this project provide evidence that Slit2

has a protective role after brain injury. We have elucidated previously unknown

dual function of Slit2 after brain injury, which includes reducing

neuroinflammation by inhibiting the migration of peripheral immune cells and anti-

permeability effects by stabilizing the BBB tight junction. Furthermore, our

findings demonstrate that the protective mechanism of Slit2 is mediated via

Robo1 receptor expressed by peripheral immune cells and Robo4 receptor

expressed by BBB endothelial cells. These novel findings show that exogenous

Slit2 administration targets two major pathophysiological events after SBI and thereby augments the body’s endogenous protective response against SBI.

Overall, these findings provide evidence that Slit2 has beneficial role after

SBI and is a potentially suitable therapeutic candidate to reduce deleterious

consequences after SBI and possibly other brain injuries with similar pathologies.

Limitations and Future Directions

Role of Other Members of the Slit Protein Remains to be Elucidated

This project primarily focused on exploring the role of Slit2 after SBI even

though there are three known isoforms of Slit proteins (Slit1-Slit3) (Guan and

Rao, 2003, Hohenester, 2008). Studies show that other Slit members may

regulate processes in the CNS too. For instance, Slit1 is predominantly

expressed in the developing CNS and Slit3 has been reported to have anti-

permeability effects following VEGF induced retinal hyperpermeability (Marillat et

al., 2002, Jones et al., 2008). Given these findings, it is likely that Slit1 and Slit3

may be involved during recovery after CNS injury. However, Hagino et al.

observed that Slit2 was predominantly expressed surrounding the lesion after

traumatic brain injury in mice while the expression of Slit1 and Slit3 was much

weaker (Hagino et al., 2003). In accordance, we observed that the expression of

Slit2 was increased after SBI. However, we did not evaluate if Slit1 and Slit3

exhibit any changes after SBI. Neither did we explore therapeutic potential of the

other Slit members after SBI. A detailed examination of all members of the Slit

family after brain injury is warranted.

Alternate Pathways in Slit2 Mediated Protection Need to be Evaluated

This project examined two primary pathways in Slit2 mediated protection

after SBI which included the Robo1-srGAP1 pathway regulating

neuroinflammation and the Robo4-paxillin pathway maintaining the endothelial

pathways, which was not explored in this project.

For instance, (1) Downstream effectors such as Abelson kinase (Abl) and

Enabled (Ena) can interact with the intracellular motif of Robo and regulate cell

migration. Abl antagonizes the repulsive effects of Robo whereas, Ena enhances

Robo mediated repulsive signaling pathway (Ballard and Hinck, 2012). (2) We

measured Cdc42 activity as the downstream substrate of srGAP1 but other small

GTPases including RhoA and Rac1 that regulate cellular actin organization and

motility were not evaluated in this project (Wojciak-Stothard and Ridley, 2002,

Waschke et al., 2004). (3) Previous studies have shown that Slit2 can inhibit

chemokine induced Src kinase activity and Lck kinase activity which have been

reported to regulate cell migration (Prasad et al., 2007). (4) The endothelial

barrier protective effect of Slit2 may be mediated by modulation of

matrixmetalloproteinase (MMP)-9 activity which was not explored (Cai et al.,

2015). (5) Lastly, using knockout or transgenic animals would strengthen the

findings in our project. It was previously observed that Robo4 null mice had

increased severity of lung injury than Robo4+/+ mice when subjected to lung

inflammation (London et al., 2010), which proposes a protective function for Slit2-

Robo4. However, Slit2 transgenic mice were more susceptible to collagenase

induced ICH and had larger hemorrhagic volumes compared to control mice

(Han and Geng, 2011). The findings in this study conflict with the protective

effects of Slit2 observed in other animal models of CNS injury (Altay et al., 2007,

Sherchan et al., 2015). This suggests that endogenous Slit2 overexpression and

endothelial cells.

Additionally, the findings in these studies could be attributed to differences

in the animal model used, differential mechanism of the endogenous versus

exogenous Slit2, or due to differences in the magnitude of exposure to Slit2 in

transgenic animals as opposed to exogenous recombinant Slit2 administration.

Although the temporal pattern and duration of Slit2 expression in transgenic

animals subjected to injury and the mechanism of injury was not explored, these

findings necessitate further exploration of the function of Slit2 after brain injury.

Detailed Exploration of the Robo Receptor Subtypes is Required

The effect of Slit2 may depend on the type of receptor predominantly

expressed following an injury. Furthermore, the expression and function of Robo

receptors may be tissue specific and context dependent. For instance, Slit2

reduced permeability in human pulmonary microvascular endothelial cells

(PMECs) that predominantly express Robo4 (Gorbunova et al., 2013), but the

anti-permeability effect of Slit2 was not observed in the human umbilical vein

endothelial cells (HUVECs) which express similar levels of Robo4 and Robo1

receptors (Gorbunova et al., 2013). Therefore, an in depth understanding of the

expression of Robo receptors and changes that may occur after SBI is required.

Upstream Regulators of Slit2 Need to be Explored

The transcription factor such as Pax6 has been shown to regulate the

for instance hypermethylation of the gene in systemic tumors and glioma have

been observed (Dallol et al., 2003). Our findings showed that endogenous Slit2

levels increased after SBI as a protective response to counteract the

consequences of SBI. However, the upstream regulators for Slit2 were not

examined. Further exploration into the regulation of endogenous Slit2 is essential

to understanding this response.

Conclusion

Our findings suggest that Slit2 has a beneficial role against

neuropathological consequences after SBI. The protective effect of Slit2 against

SBI was possibly mediated by its anti-migratory function against peripheral

leukocytes and by endothelial barrier stabilization dependent on the Robo

receptors. The dual protective function of Slit2 makes it a potential therapeutic

option to reduce neurosurgical injury and improve post-operative outcomes in

References

Altay T, McLaughlin B, Wu JY, Park TS, Gidday JM (2007) Slit modulates cerebrovascular inflammation and mediates neuroprotection against global cerebral ischemia. Experimental neurology 207:186-194.

Ballard MS, Hinck L (2012) A roundabout way to cancer. Advances in cancer research 114:187-235.

Bruder N, Ravussin P (1999) Recovery from anesthesia and postoperative extubation of neurosurgical patients: a review. Journal of neurosurgical anesthesiology 11:282-293.

Cai H, Liu W, Xue Y, Shang X, Liu J, Li Z, Wang P, Liu L, Hu Y, Liu Y (2015) Roundabout 4 regulates blood-tumor barrier permeability through the modulation of ZO-1, Occludin, and Claudin-5 expression. Journal of neuropathology and experimental neurology 74:25-37.

Dallol A, Krex D, Hesson L, Eng C, Maher ER, Latif F (2003) Frequent epigenetic inactivation of the SLIT2 gene in gliomas. Oncogene 22:4611-4616.

Gorbunova EE, Gavrilovskaya IN, Mackow ER (2013) Slit2-Robo4 receptor responses inhibit ANDV directed permeability of human lung

microvascular endothelial cells. Antiviral research 99:108-112.

Guan KL, Rao Y (2003) Signalling mechanisms mediating neuronal responses to guidance cues. Nature reviews Neuroscience 4:941-956.

Hagino S, Iseki K, Mori T, Zhang Y, Hikake T, Yokoya S, Takeuchi M, Hasimoto H, Kikuchi S, Wanaka A (2003) Slit and glypican-1 mRNAs are

coexpressed in the reactive astrocytes of the injured adult brain. Glia 42:130-138.

Han HX, Geng JG (2011) Over-expression of Slit2 induces vessel formation and changes blood vessel permeability in mouse brain. Acta pharmacologica Sinica 32:1327-1336.

Hohenester E (2008) Structural insight into Slit-Robo signalling. Biochemical Society transactions 36:251-256.

Huang L, Sherchan P, Wang Y, Reis C, Applegate RL, 2nd, Tang J, Zhang JH (2015) Phosphoinositide 3-Kinase Gamma Contributes to

Neuroinflammation in a Rat Model of Surgical Brain Injury. The Journal of neuroscience : the official journal of the Society for Neuroscience

Hyong A, Jadhav V, Lee S, Tong W, Rowe J, Zhang JH, Tang J (2008) Rosiglitazone, a PPAR gamma agonist, attenuates inflammation after surgical brain injury in rodents. Brain research 1215:218-224.

Jones CA, London NR, Chen H, Park KW, Sauvaget D, Stockton RA, Wythe JD, Suh W, Larrieu-Lahargue F, Mukouyama YS, Lindblom P, Seth P, Frias A, Nishiya N, Ginsberg MH, Gerhardt H, Zhang K, Li DY (2008) Robo4

stabilizes the vascular network by inhibiting pathologic angiogenesis and endothelial hyperpermeability. Nature medicine 14:448-453.

Kanellis J, Garcia GE, Li P, Parra G, Wilson CB, Rao Y, Han S, Smith CW, Johnson RJ, Wu JY, Feng L (2004) Modulation of inflammation by slit protein in vivo in experimental crescentic glomerulonephritis. The American journal of pathology 165:341-352.

London NR, Zhu W, Bozza FA, Smith MC, Greif DM, Sorensen LK, Chen L, Kaminoh Y, Chan AC, Passi SF, Day CW, Barnard DL, Zimmerman GA, Krasnow MA, Li DY (2010) Targeting Robo4-dependent Slit signaling to survive the cytokine storm in sepsis and influenza. Science translational medicine 2:23ra19.

Marillat V, Cases O, Nguyen-Ba-Charvet KT, Tessier-Lavigne M, Sotelo C, Chedotal A (2002) Spatiotemporal expression patterns of slit and robo genes in the rat brain. The Journal of comparative neurology 442:130-155.

Mertsch S, Schmitz N, Jeibmann A, Geng JG, Paulus W, Senner V (2008) Slit2 involvement in glioma cell migration is mediated by Robo1 receptor. Journal of neuro-oncology 87:1-7.

Prasad A, Qamri Z, Wu J, Ganju RK (2007) Slit-2/Robo-1 modulates the CXCL12/CXCR4-induced chemotaxis of T cells. Journal of leukocyte biology 82:465-476.

Rolston JD, Han SJ, Lau CY, Berger MS, Parsa AT (2014) Frequency and predictors of complications in neurological surgery: national trends from 2006 to 2011. Journal of neurosurgery 120:736-745.

Sherchan P, Huang L, Wang Y, Akyol O, Tang J, Zhang JH (2015) Recombinant Slit2 attenuates neuroinflammation after surgical brain injury by inhibiting peripheral immune cell infiltration via Robo1-srGAP1 pathway in a rat model. Neurobiology of disease 85:164-173.

Tsuchiya R, Takahashi K, Liu FC, Takahashi H (2009) Aberrant axonal

projections from mammillary bodies in Pax6 mutant mice: possible roles of Netrin-1 and Slit 2 in mammillary projections. Journal of neuroscience research 87:1620-1633.

Waschke J, Baumgartner W, Adamson RH, Zeng M, Aktories K, Barth H, Wilde C, Curry FE, Drenckhahn D (2004) Requirement of Rac activity for

maintenance of capillary endothelial barrier properties. American journal of physiology Heart and circulatory physiology 286:H394-401.

Wojciak-Stothard B, Ridley AJ (2002) Rho GTPases and the regulation of endothelial permeability. Vascular pharmacology 39:187-199.

Wong JM, Panchmatia JR, Ziewacz JE, Bader AM, Dunn IF, Laws ER, Gawande AA (2012) Patterns in neurosurgical adverse events: intracranial neoplasm surgery. Neurosurgical focus 33:E16.

Ye BQ, Geng ZH, Ma L, Geng JG (2010) Slit2 regulates attractive eosinophil and repulsive neutrophil chemotaxis through differential srGAP1 expression during lung inflammation. Journal of immunology 185:6294-6305.